A patient with Huntington's disease and long-surviving fetal neural transplants that developed mass lesions

被引:81
作者
Keene, C. Dirk [1 ]
Chang, Rubens C. [1 ]
Leverenz, James B. [2 ]
Kopyov, Oleg [5 ]
Perlman, Susan [6 ]
Hevner, Robert F. [1 ,3 ,4 ]
Born, Donald E. [1 ]
Bird, Thomas D. [2 ]
Montine, Thomas J. [1 ]
机构
[1] Univ Washington, Med Ctr, Harborview Med Ctr, Dept Pathol,Div Neuropathol, Seattle, WA 98104 USA
[2] Univ Washington, Med Ctr, Dept Neurol, VA Puget Sound Hlth Care Syst, Seattle, WA 98104 USA
[3] Univ Washington, Med Ctr, Dept Neurosurg, Seattle Childrens Hosp, Seattle, WA 98104 USA
[4] Reg Med Ctr, Seattle, WA 98104 USA
[5] St Johns Reg Med Ctr, Inst Neurosci, Oxnard, CA 93030 USA
[6] Univ Calif Los Angeles, Dept Neurol, Los Angeles, CA 90095 USA
关键词
Huntington's disease; Human fetal neural transplantation; Graft survival and differentiation; Transplant overgrowth; Peripheral nerve co-graft; AUTOLOGOUS ADRENAL-MEDULLA; PARKINSONS-DISEASE; INTRASTRIATAL COGRAFTS; PERIPHERAL-NERVE; FOLLOW-UP; ALLOGRAFTS; PATHOLOGY; SAFETY; NEUROTRANSPLANTATION; STRIATUM;
D O I
10.1007/s00401-008-0465-0
中图分类号
R74 [神经病学与精神病学];
学科分类号
100204 [神经病学];
摘要
Transplantation of human fetal neural tissue into adult neostriatum is an experimental therapy for Huntington's disease (HD). Here we describe a patient with HD who received ten intrastriatal human fetal neural transplants and, at one site, an autologous sural nerve co-graft. Although initially clinically stable, she developed worsening asymmetric upper motor neuron symptoms in addition to progression of HD, and ultimately died 121 months post transplantation. Eight neural transplants, up to 2.9 cm, and three ependymal cysts, up to 2.0 cm, were identified. The autologous sural nerve co-graft was found adjacent to the largest mass lesion, which, along with the ependymal cyst, exhibited pronounced mass effect on the internal capsules bilaterally. Grafts were composed of neurons and glia embedded in disorganized neuropil; robust Y chromosome labeling was present in a subset of grafts and cysts. The graft-host border was discrete, and there was no evidence of graft rejection or HD pathologic changes within donor neurons. This report, for the first time, highlights the potential for graft overgrowth in a patient receiving fetal neural transplantation.
引用
收藏
页码:329 / 338
页数:10
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